Kwski Medicl Journl 40(2):79-85,2014 doi:10.11482/kmj-e40(2)79 79 A cse report of isolted prescrl squmous cell crcinom developed four yers fter gstrectomy Kori SHIGEMITSU, Noms ISHIDA, Yoko HIRABAYASHI, Munenori TAKAOKA, Jiro HAYASHI, Tkuy FUKAZAWA, Kzuhiro YOSHIDA, Atsushi URAKAMI, Tomoki YAMATSUJI, Kzutk NAKASHIMA, Ichiro MORITA, Minoru HAISA, Yoshio NAOMOTO Deprtment of Generl Surgery, Kwski Hospitl, Kwski Medicl School, 2-1-80 Nksnge, Kit-ku, Okym 700-8505, Jpn ABSTRACT Chemordition therpy nd trnsscrl resection were performed to tret isolted squmous cell crsinom tht occurred in prescrl tissues in the pelvis four yers fter gstrectomy due to erly gstric cncer, with the prognosis continuing to e fvorle. The ptient ws 57-yer-old womn, who cme to our hospitl hving symptoms of nemi four yers fter gstrectomy. After rectl exmintion, tumor mss the size of sprrowegg ws discovered on the left rectl wll. An dominl CT showed tumor, 3.7cm 3.7cm in size, on the outer left wll of the upper rectum. After CT-guided iopsy, squmous cell crcinom ws detected. Irrdition (totl 40 Gy) nd chemotherpy (MMC+5-FU) were performed, mss shrinkge ws confirmed, nd trnsscrl tumor resection ws performed. According to the histopthologicl exmintion, very smll ut vile cncer ws found to e remining. 4 yers fter the tumor removl, no recurrence hs een discovered. Squmous cell crcinom in the pelvis often origintes from the vgin. However, the ptient didn t hve ny mlignnt findings from genitl exmintion t the time the symptoms ppered, nd this cse ws dignosed s isolted squmous cell crcinom. A trnsscrl pproch to remove such tumor is considered to e useful ecuse it is reltively low invsion nd preserves nl functions. doi:10.11482/kmj-e40(2)79 (Accepted on Novemer 12, 2014) Key words: Prescrl tumor, Squmous cell crcinom, Trnsscrl resection, Epidermoid cyst INTRODUCTION Mlignnt tumors in the prescrl spce often originte from the rectum, uterus, dnex, vgin, ldder, or hve metstsized from tumor in nother prt of the ody. This is cse report of squmous cell crcinom developed in the prescrl spce four yers fter gstrectomy due to erly gstric cncer. The cncer ws treted y chemordition therpy nd trnsscrl resection. Corresponding uthor Kori Shigemitsu Deprtment of Generl Surgery, Kwski Hospitl, Kwski Medicl School, 2-1-80 Nksnge, Kit-ku, Okym 700-8505, Jpn Phone : 81 86 225 2111 Fx : 81 86 232 8343 E-mil: nmste@med.kwski-m.c.jp
80 Kwski Medicl Journl CASE REPORT A 57 yer-old femle who hd undergone distl gstrectomy for gstric cncer (in the ody of stomch, poorly differentited denocrcinom, sm2, no lymph node metstsis) four yers go visited our hospitl for nemi. She hd received follow-up tretments fter the gstrectomy, ut y her own decision she stopped receiving the tretments three yers fter the gstrectomy. In Mrch of the fourth yer fter her opertion, she felt lightheded nd sw nery doctor. In April, she cme to our hospitl to hve more detiled exmintion. No normlities were found in either the hed nd neck re or the chest. No superficil lymph node ws enlrged. The domen ws flt nd soft, with no norml owel sound. A tumor mss the size of sprrow egg (1.5-2 cm) ws detected y digitl rectl exmintion on the outside of the left wll of the rectum. Blood exmintion reveled no remrkle normlities except moderte nemi. CEA, CA19-9 nd SCC were within the norml rnges. Adominl CT showed tumor mss pproximtely 3.7 cm 3.7 cm in size, with necrosis in it, on the outside of the left rectum ove the peritonel reflection. The rim of the tumor mss ws modertely enhnced y dynmic contrstenhnced CT (Fig. 1). The tumor mss ws touching the uterine corpus, the rectum ove the peritonel reflection nd the pelvic wll. Invsion couldn t e ruled out, ut dditionl ovious metstsis to other prts of the ody including the lymph nodes ws not found. CTs from the time of her stomch cncer 4 yers prior were re-exmined retrospectively nd nodulr shdow of low density pproximtely 1.8 1.2 cm ws found in the sme re. Pelvic MRI reveled the lesion ws prtilly djcent to the uterine corpus nd posterior vginl fornix, ut it ws not successive nd oth ovries were norml. However, the lesion ws strongly dhered to the rectum ove the peritonel reflection nd the left nterior scrum, nd severe dhesion nd/or invsion ws suspected (Fig. 2). Upper gstrointestinl c Fig.1. Dynmic contrst-enhnced CT(Yellow rrows show the modertely enhnced tumor) : Axil view reveled the tumor ws touching the uterine corpus nd rectum. : Axil view reveled the tumor ws touching the scrum. c: Sgittl view reveled the tumor ws touching the uterine corpus.
Shigemitsu K, et l. : prescrl squmous cell crcinom 81 Fig.2. Pelvic MRI (Yellow rrows show the prescrl tumor) : The tumor ws prtilly djcent to the uterine corpus nd posterior vginl fornix, ut it ws not successive nd oth ovries were norml. : The tumor ws strongly dhered to the rectum nd the left nterior scrum, nd severe dhesion nd/ or invsion ws suspected. c: Sgittl view reveled the tumor dhered to the rectum ove the peritonel reflection. c Fig.3. : CT-guided needle iopsy ws performed using 20 G cutting needle plced in the prone position. : Squmous cell crcinom ws detected fter histopthologicl exmintion. endoscopy showed no significnt normlities in either the remining stomch or duodenum. Colorectl endoscopy showed dolichocolon. A 3 cm-sized mild ulge tht seemed to e cused y the pressure on the outer wll ws found on the left nterior wll of the rectum ove the peritonel reflection, 10 cm ove the nl verge. Vginl exfolitive cytodignosis reveled no mlignnt finding on the vginl mucous memrne. CT-guided needle iopsy ws performed using 20 G cutting needle plced in the prone position (Fig. 3). Squmous cell crcinom ws detected
82 Kwski Medicl Journl fter histopthologicl exmintion (Fig. 3). The lesion ws dignosed s squmous cell crcinom originting from the pelvis. Irrdition (10MV x-ry, 2 Gy prllel opposing portls per tretment, 5 times week, totl 40 Gy) nd chemotherpy (MMC 100 mg/m 2, D1+5-FU 1,000 mg/m 2, D1-4) were strted sed on stndrd nl cnl cncer tretment. A month nd two weeks fter chemordition ws finished, the tumor ws confirmed y dominl CT to hve shrunk to 1.2 cm. 2 months fter chemordition ws finished, trnsscrl resection ws performed. With the ptient in the prone jckknife position nd the uttocks were tped prt, the procedure strted with n ngled incision from the left of the fourth scrl verter, through the tip of the coccyx, to 3 cm wy from the nl verge. Then the gluteus mximus muscle ws resected t the point of connection of oth sides of the coccyx. The coccyx ws resected t its hed (Fig. 4) nd the nococcygel rche ws resected s well. A tumor mss the size of chicken egg tht ws fixed to the left wll of the rectum ws found digitlly. The uppermost portion of the tumor mss ws dhered to the peritonel reflection so the pelvic cvity nd the dominl cvity were opened t the time of removl. Dorslly, while leeding from the venous plexus in the nterior scrum ws stopped, nd the tumor ws removed from the surrounding tissues, leving only its connection to the invded rectl wll. We resected the mesorectum round the invded re nd exposed the rectl dventiti. Then with n electric sclpel, we resected the dventiti nd musculris propri nd exposed the sumucos. Using n utomtic suturing device, we resected the invded prt of the rectl wll nd removed the tumor long with the rectl wll (Fig. 4). A smple of the resection stump ws tken to frozen section pthology nd its mlignncy ws confirmed negtive. Histopthologicl exmintion reveled hylinized connective tissue mong the pelvic tissue hd incresed, nd cornified cells tht lpsed into necrosis in the connective tissues were lso found. There ws fom cell infiltrtion round the cornified cells (Fig. 5). Isolted nd scttered vesicles of dysplstic cells were found nd very little yet vile squmous cncer remined (Fig. 5). Immunostining ws p16-positive (Fig. 5c). The rdiotherpy tretment effectiveness s mesured y histopthologicl criteri ws Grde 2. No tumorcell invsion in the rectum ws found histologiclly. The postopertive course ws uneventful, nd the ptient ws dischrged 14 dys fter the opertion. She is receiving out-ptient follow-up tretments nd no reoccurrence hs een found fter 4 yers. Fig.4. Opertive findings : The coccyx ws resected t its hed. : Using n utomtic suturing device, we resected the invded prt of the rectl wll nd removed the tumor long with the rectl wll.
Shigemitsu K, et l. : prescrl squmous cell crcinom 83 c Fig.5. Histopthologicl findings : The fom cell infiltrtion round the cornified cells. : Isolted nd scttered vesicles of dysplstic cells were found nd very little yet vile squmous cncer remined. c: Immunostining reveled p16 ws positive. DISCUSSION The prescrl spce is ounded nteriorly y the rectum, posteriorly y the scrum nd coccyx, superiorly y the peritonel reflection (generlly elow the S2 level), nd inferiorly y the pelvic floor muscles. This re is cudl end, the site of fusion of the emryologic hindgut, neuroectoderm nd contins mny types of tissue nd totipotentil cells tht cn led to the development of vrious tumors; neurofirosrcom, neurilemmom, leiomyom, chordom, tertoid, lipom, desmoids, GIST (Gstrointestinl stroml tumor) nd metsttic tumors. Although the true incidence of such tumors is unknown, it is estimted t one in every 40,000 hospitl dmissions 1,2). In generl, they re symptomtic over prolonged periods nd re often discovered incidently 3,4). Digitl rectl exmintion is very importnt for dignosis of prescrl tumors, Testini et l. 5) reported tht 76 % - 90 % of ptients presented with plple tumors in the digitl rectl exmintion. CT nd/or MRI hve ecome the est dignostic modlities for prescrl tumors. CT cn e used to ssess size, upper extent, ehvior, nd whether surrounding structures re involved 6). MRI is highly recommended in prescrl tumors, ecuse of its multiplnr cpcity nd prticulrly useful in delineting soft-tissue plnes nd ony nd nerve invsion 7). Generlly, it is sid tht needle iopsy for prescrl tumors should e voided ecuse complictions including meningitis fter punction of meningocele, infection of cystic tumor, mlignnt extension through the iopsy trct or leeding cn occur 8). But in our cse, histologicl dignosis ws required to distinguish whther originting or metsttic tumor. In this cse, the ptient hd hd stomch cncer in the pst. However, the cncer ws t n erly stge nd histologiclly clssified s denocrcinom.
84 Kwski Medicl Journl Therefore, the squmous cell crcinom ws not likely metsttisized from the stomch cncer. We could find no report of squmous cell crcinom originting from the prescrl spce, other thn mlignnt regenertion of epidermoid cyst, which is very rre. Hwkins et l. 9) recognized tht cystic tumor msses were generted s developmentl cyst y developmentl error during the fetl stge, nd mde 3 histologicl clssifictions: dermoid cysts, epidermoid cysts nd mucus secreting cysts. Among the three, epidermoid cysts consist of strtified squmous epitheli tht do not include cutneous ppendges, nd there re lso some reports tht the msses turned mlignnt 10,11). Moreover, our ptient s cyst from 4 yers go showed low density, mening it ws cystic node. Therefore, it cn e sid tht there is high possiility tht n epidermoid cyst ws the cuse of the tumor in this cse. Also, immunostining for the tumor from this ptient ws p16-positive. p16 is chrcteristiclly positive when there is high-grde squmous intrepithelil lesion (HSIL) relted to n infection of high-risk humn ppillomvirus in the uterine cervix, or when there is n denocrcinom in situ (AIS). This polity ws found from the thorough gynecologicl exmintions efore nd fter the surgery. Thus, the psitive result is considered significnt when clssifying rective squmous typi or not. No normtient ws dignosed with isolted squmous cncer derived from the tissues in the vginl portion of the cervix, not metsttic lesion. Excision of prescrl tumor is essentil, even in symptomtic ptients for the possiility of mlignncy, potentil dystoci in women of childering ge, future mlignnt trnsformtion or infection. The four min pproches to excision of prescrl tumors re s follows: the trnsscrl pproch with or without coccygel excision, the trnsdominl pproch, the comined trnsscrl/ trnsdominl pproch, nd the trnsrectl pproch. For erly-stge cncer in the rectum elow the peritonel reflection, trns-scrl pproch for locl rectl excision cn e performed to preserve nl sphincter functions. As stted erlier, isolted prescrl squmous cell crcinom is rre nd there is no stndrd surgicl procedure. The surgicl pproch nd extent of resection for prescrl lesion is determined y the loction, size nd its mlignnt potentil while tking into ccount whether or not the scrum or djcent viscer is involved 12). Loclio 13) suggested tht when treting prescrl tumor, trnsscrl pproch should e chosen when tumor is enign nd smller thn 8 cm, nd n dominoscrl pproch should e chosen when the tumor hs lrger dimeter or is mlignnt nd could cuse n infection. Recently, some reports reveled if the tumor is positioned elow the level of the S3 vertere, trnsscrl pproch cn e considered. But tumors tht extend ove S3 require n dominl or comined pproch 12,14). In our cse, irrdition (10MV x-ry, 2 Gy prllel opposing portls per tretment, 5 times week, totl 40 Gy) nd chemotherpy (MMC 100 mg/ m2, D1+5-FU 1,000 mg/m2, D1-4) were strted sed on stndrd tretment for nl cnl cncer, which is generlly squmous cell crcinom 15,16). Metstsis to neighoring lymph nodes ws not found efore the surgery, nd pprent shrinkge ws recognized fter the rdiotherpy ut efore the surgery. Therefore, removing the tumor ws possile ecuse the resection stump ws negtive, including the rectl dventiti tht trns-scrlly dhered to the tumor. Bowel movement function fter the surgery ws good nd currently there is no sign of recurrence or metstsis including in the locl res. Therefore, it cn e concluded tht the pproch in this report to tret isolted prescrl squmous cell crcinom ws effective. REFERENCES 1)Whittker LD, Pemerton JD. Tumors ventrl to the
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